Validation of a modified Care and Comfort Score and responsiveness to hip surgery in children with cerebral palsy in Gross Motor Function Classification System levels IV and V.

Aims: The purpose of this study was to assess the reliability and responsiveness to hip surgery of a four-point modified Care and Comfort Hypertonicity Questionnaire (mCCHQ) scoring tool in children with cerebral palsy (CP) in Gross Motor Function Classification System (GMFCS) levels IV and V. Methods: This was a population-based cohort study in children with CP from a national surveillance programme. Reliability was assessed from 20 caregivers who completed the mCCHQ questionnaire on two occasions three weeks apart. Test-retest reliability of the mCCHQ was calculated, and responsiveness before and after surgery for a displaced hip was evaluated in a cohort of children. Results: Test-retest reliability for the overall mCCHQ score was good (intraclass correlation coefficient 0.78), and no dimension demonstrated poor reliability. The surgical intervention cohort comprised ten children who had preoperative and postoperative mCCHQ scores at a minimum of six months postoperatively. The mCCHQ tool demonstrated a significant improvement in overall score from preoperative assessment to six-month postoperative follow-up assessment (p < 0.001). Conclusion: The mCCHQ demonstrated responsiveness to intervention and good test-retest reliability. The mCCHQ is proposed as an outcome tool for use within a national surveillance programme for children with CP.

Responsiveness of the Foot Profile Score in children with hemiplegia.

BACKGROUND: The Foot Profile Score (FPS) is a single score that summarises foot posture and dynamic foot motion during the gait cycle based on the kinematic data of the Oxford Foot Model. The FPS enables clinicians and researchers to quantify foot abnormalities during gait, to monitor change in foot/ankle motion over time, and to measure the outcome of intervention. With the creation of a new outcome measure, it is important to test its responsiveness in a clinical population for whom it may be sensitive to change. AIM: To evaluate the responsiveness of the FPS in a clinical population following isolated foot and ankle surgery. METHODS: Using previous work completed to validate the FPS, we defined the minimal clinically important difference (MCID) for the FPS. Using this MCID, we applied it to a clinical population of 37 children with cerebral palsy, spastic hemiplegia, comparing their FPS before and after foot and ankle surgery. A regression analysis looked at potential relationships between the change in FPS and their pre-operative FPS, age at surgery, and time since surgery. RESULTS: An MCID of 2.4 degrees was calculated through regression analysis. The mean change from the pre-operative FPS to the post-operative FPS was 4.6 (SD 3.7 with a range from -0.1 to 13.4). Twenty-eight children (76%) had a change in their FPS greater than the MCID. A regression analyses only showed a clear regression between pre-operative FPS and change in FPS (R2 = 0.58 p < 0.01).

Reliability testing of the heel marker in three-dimensional gait analysis.

INTRODUCTION: In three-dimensional gait analysis, anatomical axes are defined by and therefore sensitive to marker placement. Previous analysis of the Oxford Foot Model (OFM) has suggested that the axes of the hindfoot are most sensitive to marker placement on the posterior aspect of the heel. Since other multi-segment foot models also use a similar marker, it is important to find methods to place this as accurately as possible. The aim of this pilot study was to test two different 'jigs' (anatomical alignment devices) against eyeball marker placement to improve reliability of heel marker placement and calculation of hindfoot angles using the OFM. METHODS: Two jigs were designed using three-dimensional printing: a ratio caliper and heel mould. OFM kinematics were collected for ten healthy adults; intra-tester and inter-tester repeatability of hindfoot marker placement were assessed using both an experienced and inexperienced gait analyst for 5 clinically relevant variables. RESULTS: For 3 out of 5 variables the intra-tester and inter-tester variability was below 2 degrees for all methods of marker placement. The ratio caliper had the lowest intra-tester variability for the experienced gait analyst in all 5 variables and for the inexperienced gait analyst in 4 out of 5 variables. However for inter-tester variability, the ratio caliper was only lower than the eyeball method in 2 out of the 5 variables. The mould produced the worst results for 3 of the 5 variables, and was particularly prone to variability when assessing average hindfoot rotation, making it the least reliable method overall. CONCLUSIONS: The use of the ratio caliper may improve intra-tester variability, but does not seem superior to the eyeball method of marker placement for inter-tester variability. The use of a heel mould is discouraged.

A comparison of conventional and minimally invasive multilevel surgery for children with diplegic cerebral palsy.

AIMS: To compare changes in gait kinematics and walking speed 24 months after conventional (C-MLS) and minimally invasive (MI-MLS) multilevel surgery for children with diplegic cerebral palsy (CP). METHODS: A retrospective analysis of 19 children following C-MLS, with mean age at surgery of 12 years five months (seven years ten months to 15 years 11 months), and 36 children following MI-MLS, with mean age at surgery of ten years seven months (seven years one month to 14 years ten months), was performed. The Gait Profile Score (GPS) and walking speed were collected preoperatively and six, 12 and 24 months postoperatively. Type and frequency of procedures as part of MLS, surgical adverse events, and subsequent surgery were recorded. RESULTS: In both groups, GPS improved from the preoperative gait analysis to the six-month assessment with maintenance at 12 and 24 months postoperatively. While reduced at six months in both groups, walking speed returned to preoperative speed by 12 months. The overall pattern of change in GPS and walking speed was similar over time following C-MLS and MI-MLS. There was a median of ten procedures per child as part of both C-MLS (interquartile range (IQR) 8.0 to 11.0) and MI-MLS (IQR 7.8 to 11.0). Surgical adverse events occurred in seven (37%) and 13 (36%) children, with four (21%) and 13 (36%) patients requiring subsequent surgery following C-MLS and MI-MLS, respectively. CONCLUSION: This study indicates similar improvements in gait kinematics and walking speed 24 months after C-MLS and MI-MLS for children with diplegic CP. Cite this article: Bone Joint J 2021;103-B(1):192-197.

Foot function during gait and parental perceived outcome in older children with symptomatic club foot deformity.

AIMS: To assess if older symptomatic children with club foot deformity differ in perceived disability and foot function during gait, depending on initial treatment with Ponseti or surgery, compared to a control group. Second aim was to investigate correlations between foot function during gait and perceived disability in this population. METHODS: In all, 73 children with idiopathic club foot were included: 31 children treated with the Ponseti method (mean age 8.3 years; 24 male; 20 bilaterally affected, 13 left and 18 right sides analyzed), and 42 treated with primary surgical correction (mean age 11.6 years; 28 male; 23 bilaterally affected, 18 left and 24 right sides analyzed). Foot function data was collected during walking gait and included Oxford Foot Model kinematics (Foot Profile Score and the range of movement and average position of each part of the foot) and plantar pressure (peak pressure in five areas of the foot). Oxford Ankle Foot Questionnaire, Disease Specific Index for club foot, Paediatric Quality of Life Inventory 4.0 were also collected. The gait data were compared between the two club foot groups and compared to control data. The gait data were also correlated with the data extracted from the questionnaires. RESULTS: Our findings suggest that symptomatic children with club foot deformity present with similar degrees of gait deviations and perceived disability regardless of whether they had previously been treated with the Ponseti Method or surgery. The presence of sagittal and coronal plane hindfoot deformity and coronal plane forefoot deformity were associated with higher levels of perceived disability, regardless of their initial treatment. CONCLUSION: This is the first paper to compare outcomes between Ponseti and surgery in a symptomatic older club foot population seeking further treatment. It is also the first paper to correlate foot function during gait and perceived disability to establish a link between deformity and subjective outcomesCite this article: Bone Joint Open 2020;1-7:384-391.

What is the functional mobility and quality of life in patients with cerebral palsy following single-event multilevel surgery?

PURPOSE: To report functional mobility in patients with diplegic cerebral palsy (CP) at long-term follow-up after single-event multilevel surgery (SEMLS). The secondary aim was to assess the relationship between functional mobility and quality of life (QoL) in patients previously treated with SEMLS. METHODS: A total of 61 patients with diplegic CP, mean age at surgery 11 years, eight months (sd 2 years, 5 months), were included. A mean of eight years (sd 3 years, 10 months) after SEMLS, patients were contacted and asked to complete the Functional Mobility Scale (FMS) questionnaire over the telephone and given a weblink to complete an online version of the CP QOL Teen. FMS was recorded for all patients and CP QOL Teen for 23 patients (38%). RESULTS: Of patients graded Gross Motor Function Classification System (GMFCS) I and II preoperatively, at long-term follow-up the proportion walking independently at home, school/work and in the community was 71% (20/28), 57% (16/28) and 57% (16/28), respectively. Of patients graded GMFCS III preoperatively, at long-term follow-up 82% (27/33) and 76% (25/33) were walking either independently or with an assistive device at home and school/work, respectively, while over community distances 61% (20/33) required a wheelchair. The only significant association between QoL and functional mobility was better 'feelings about function' in patients with better home FMS scores (r = 0.55; 95% confidence interval 0.15 to 0.79; p = 0.01). CONCLUSION: The majority of children maintained their preoperative level of functional mobility at long-term follow-up after SEMLS. LEVEL OF EVIDENCE: IV.

Two-tailed significance tests for 2 × 2 contingency tables: What is the alternative?

Two-tailed significance testing for 2 × 2 contingency tables has remained controversial. Within the medical literature, different tests are used in different papers and that choice may decide whether findings are adjudged to be significant or nonsignificant; a state of affairs that is clearly undesirable. In this paper, it is argued that a part of the controversy is due to a failure to recognise that there are two possible alternative hypotheses to the Null. It is further argued that, while one alternative hypothesis can lead to tests with greater power, the other choice is more applicable in medical research. That leads to the recommendation that, within medical research, 2 × 2 tables should be tested using double the one-tailed exact probability from Fisher's exact test or, as an approximation, the chi-squared test with Yates' correction for continuity.

Validation of the foot profile score.

BACKGROUND: There are numerous static measures of foot posture but there is no published score of dynamic foot motion. Three-dimensional gait analysis can include a multi-segment foot model like the Oxford Foot Model (OFM) to comprehensively quantify foot kinematic deviations across the gait cycle but it lacks an overall score, like the Gait Profile score (GPS), used to summarize the quality of lower extremity motion. RESEARCH QUESTION: This paper introduces the Foot Profile Score (FPS), a single number, analogous to the GPS but based on kinematic data of the OFM. The aim of this study is to validate the FPS by studying its properties and design, and analyse it against a clinical assessment of foot deformity. METHODS: Concurrent validity was established for the FPS analysing the relationship with Clinical Foot Deformity Score (CFDS) in 60 subjects with a condition affecting the lower limbs globally Content validity was established for the six Foot Variable Scores (FVS) that make up the FPS using a multiple regression of the CFDS on the 6 FVS in the 60 subjects. Predictive validity was established analysing the relationship of the FPS and GPS comparing 60 global involvement subjects with 60 subjects with isolated foot deformity. RESULTS: Pearson correlation between the FPS and CFDS was significant at 0.62 (p < 0.001). Each element of FVS contributes positively to predicting the CFDS with R2 = 0.456 (p < 0.001). FPS contributed independently to the prediction of CFDS (t = 3.9, p < 0.001). The correlation between the GPS and FPS in the global involvement group was significant at r = 0.64 (p < 0.001), while there was no correlation found with r = 0.08 (p = 0.54) in the foot deformity group. SIGNIFICANCE: The FPS is the first validated score of dynamic foot motion.

Mercaptopurine versus placebo to prevent recurrence of Crohn's disease after surgical resection (TOPPIC): a multicentre, double-blind, randomised controlled trial.

BACKGROUND: Up to 60% of patients with Crohn's disease need intestinal resection within the first 10 years of diagnosis, and postoperative recurrence is common. We investigated whether mercaptopurine can prevent or delay postoperative clinical recurrence of Crohn's disease. METHODS: We did a randomised, placebo-controlled, double-blind trial at 29 UK secondary and tertiary hospitals of patients (aged >16 years in Scotland or >18 years in England and Wales) who had a confirmed diagnosis of Crohn's disease and had undergone intestinal resection. Patients were randomly assigned (1:1) by a computer-generated web-based randomisation system to oral daily mercaptopurine at a dose of 1 mg/kg bodyweight rounded to the nearest 25 mg or placebo; patients with low thiopurine methyltransferase activity received half the normal dose. Patients and their carers and physicians were masked to the treatment allocation. Patients were followed up for 3 years. The primary endpoint was clinical recurrence of Crohn's disease (Crohn's Disease Activity Index >150 plus 100-point increase in score) and the need for anti-inflammatory rescue treatment or primary surgical intervention. Primary and safety analyses were by intention to treat. Subgroup analyses by smoking status, previous thiopurines, previous infliximab or methotrexate, previous surgery, duration of disease, or age at diagnosis were also done. This trial is registered with the International Standard Randomised Controlled Trial Register (ISRCTN89489788) and the European Clinical Trials Database (EudraCT number 2006-005800-15). FINDINGS: Between June 6, 2008, and April 23, 2012, 240 patients with Crohn's disease were randomly assigned: 128 to mercaptopurine and 112 to placebo. All patients received at least one dose of study drug, and no randomly assigned patients were excluded from the analysis. 16 (13%) of patients in the mercaptopurine group versus 26 (23%) patients in the placebo group had a clinical recurrence of Crohn's disease and needed anti-inflammatory rescue treatment or primary surgical intervention (adjusted hazard ratio [HR] 0·54, 95% CI 0·27-1·06; p=0·07; unadjusted HR 0·53, 95% CI 0·28-0·99; p=0·046). In a subgroup analysis, three (10%) of 29 smokers in the mercaptopurine group and 12 (46%) of 26 in the placebo group had a clinical recurrence that needed treatment (HR 0·13, 95% CI 0·04-0·46), compared with 13 (13%) of 99 non-smokers in the mercaptopurine group and 14 (16%) of 86 in the placebo group (0·90, 0·42-1·94; pinteraction=0·018). The effect of mercaptopurine did not significantly differ from placebo for any of the other planned subgroup analyses (previous thiopurines, previous infliximab or methotrexate, previous surgery, duration of disease, or age at diagnosis). The incidence and types of adverse events were similar in the mercaptopurine and placebo groups. One patient on placebo died of ischaemic heart disease. Adverse events caused discontinuation of treatment in 39 (30%) of 128 patients in the mercaptopurine group versus 41 (37%) of 112 in the placebo group. INTERPRETATION: Mercaptopurine is effective in preventing postoperative clinical recurrence of Crohn's disease, but only in patients who are smokers. Thus, in smokers, thiopurine treatment seems to be justified in the postoperative period, although smoking cessation should be strongly encouraged given that smoking increases the risk of recurrence. FUNDING: Medical Research Council.

Thrombocytopenia and platelet transfusion in UK critical care: a multicenter observational study.

BACKGROUND: Platelet (PLT) transfusions are widely used, but few studies have described patterns of use in critical care. STUDY DESIGN AND METHODS: As part of a prospective multicenter observational study of all sequentially admitted patients to UK general intensive care units (ICUs) over 8 weeks, daily data were collected throughout admission on frequency of thrombocytopenia and use of PLT transfusions, in addition to clinical outcomes, including bleeding. RESULTS: There were 1923 admissions recruited across 29 ICUs for analysis (96.6% of all eligible admissions). The period prevalences of severe thrombocytopenia (<50 × 10(9) /L) for the entire ICU stay were 12.4% (234/1881) and 13.7% (263/1914) when the 24 hours before admission was also included. A total of 35.4% of patients who experienced severe thrombocytopenia died in the ICU. A total of 169 patients (9% of study population) received 534 units of transfused PLTs (median number of units per patient admission was 2; interquartile range, 1-3; maximum, 38). Pretransfusion PLT counts were more than 50 × 10(9) for 40% of PLT transfusions overall, and even when no clinically significant bleeding was recorded on the day of transfusion, the lowest recorded PLT count was more than 50 × 10(9) for 34% of transfusions. There was evidence of only modest increments in PLT count. CONCLUSION: Thrombocytopenia is common in critical care, but there is wide variation in PLT transfusion use. Patients commonly received PLT transfusions on days without clinically significant hemorrhage. The high prevalence of thrombocytopenia in the critically ill population and inconsistent patterns of PLT transfusions indicate the importance of improving the evidence base for PLT use.

Lies, damn lies and statistics: errors and omission in papers submitted to INJURY 2010-2012.

INTRODUCTION: Many reviews of published papers in the medical literature have reported errors in statistical methods and presentation. METHODS: 100 successive papers submitted to INJURY and sent for initial statistical review between December 2010 and January 2012 were analysed. The comments made on the papers were categorised and summarised. RESULTS: Suggestions for improvement were made for 90 of the papers. An inappropriate analysis was identified in 47. Other errors were seen in 45 papers including 9 wrong p-values for the method used. Simple numerical mistakes were common (19%). An inadequate description of some element of the study was a problem in 22 papers and additional limitations to be described in Discussion were recommended in 26. Numerically most comments were made about some element of the presentation of results. DISCUSSION: Many of the errors identified are easily avoided. Guidance on some common issues is presented. CONCLUSIONS: Statistical and numerical errors are common in papers submitted to INJURY and requiring statistical review. Following the advice in Discussion and using reporting guidelines should reduce the number of papers requiring corrections.